Specificity: A Phenotypic Comparison of Communication-Relevant Domains Between Youth With Down Syndrome and Fragile X Syndrome.

作者: Laura del Hoyo Soriano , Angela John Thurman , Leonard Abbeduto

DOI: 10.3389/FGENE.2018.00424

关键词: Intellectual disabilityFragile X syndromeCognitionDown syndromePragmaticsPhenotypeNonverbal communicationCognitive skillDevelopmental psychologyPsychology

摘要: Despite the shared presence of an intellectual disability (ID), there is a growing literature documenting important phenotypic differences between Down syndrome (DS) and fragile X (FXS). These conclusions, however, are based on synthesis across studies, each which typically includes only measures limited number constructs, with differing participant characteristics. Firmer conclusions regarding specific phenotypes require single comprehensive multi-domain assessment participants groups being well matched chronological age (CA) cognitive functioning. The current study was designed to fill this gap by assessing several behavioral domains relevant communication, such as: structural language skills, false belief understanding, as pragmatics difficulties, in 30 adolescents both sexes DS 39 males FXS, CA nonverbal (NV) cognition. After statistically controlling for NV cognition, we did not find significant expressive receptive or understanding. In contrast, displayed less stereotyped fewer difficulties compared FXS. Within-syndrome associations among targeted described. Finally, females were impaired than almost all domains, whereas no sex-related observed pragmatics, behavior. Clinical methodological implications findings discussed.

参考文章(96)
Joav Merrick, Isack Kandel, Gideon Vardi, Adolescents with Down syndrome. International journal of adolescent medicine and health. ,vol. 16, pp. 13- 20 ,(2004) , 10.1515/IJAMH.2004.16.1.13
Rosemary Tannock, Mothers' directiveness in their interactions with their children with and without Down syndrome. American Journal on Mental Retardation. ,vol. 93, pp. 154- ,(1988)
Susan M. Rivera, Allan L. Reiss, From genes to brain to behavior: the case of fragile X syndrome Neuroimaging in Developmental Clinical Neuroscience. pp. 217- 228 ,(2009) , 10.1017/CBO9780511757402.017
Susana de Sola, Rafael de la Torre, Gonzalo Sánchez-Benavides, Bessy Benejam, Aida Cuenca-Royo, Laura Del Hoyo, Joan Rodríguez, Silvina Catuara-Solarz, Judit Sanchez-Gutierrez, Ivan Dueñas-Espin, Gimena Hernandez, Jordi Peña-Casanova, Klaus Langohr, Sebastia Videla, Henry Blehaut, Magi Farre, Mara Dierssen, TESDAD Study Group, None, A new cognitive evaluation battery for Down syndrome and its relevance for clinical trials. Frontiers in Psychology. ,vol. 6, pp. 708- 708 ,(2015) , 10.3389/FPSYG.2015.00708
Carl Feinstein, Allan L. Reiss, Autism: The Point of View from Fragile X Studies. Journal of Autism and Developmental Disorders. ,vol. 28, pp. 393- 405 ,(1998) , 10.1023/A:1026000404855
Bruce J Tonge, Stewart L Einfeld, Psychopathology and Intellectual Disability: The Australian Child to Adult Longitudinal Study International Review of Research in Mental Retardation. ,vol. 26, pp. 61- 91 ,(2003) , 10.1016/S0074-7750(03)01002-4
F. Maas, Jill B. Nuccio, Leonard Abbeduto, Glenis Benson, Katherine Short, Development of a Theory of Mind in Individuals with Mental Retardation. American Journal on Mental Retardation. ,vol. 98, pp. 427- 433 ,(1993)
Donald B. Bailey, Jr., Deborah D. Hatton, Gary Mesibov, Nicole Ament, Martie Skinner, Early Development, Temperament, and Functional Impairment in Autism and Fragile X Syndrome Journal of Autism and Developmental Disorders. ,vol. 30, pp. 49- 59 ,(2000) , 10.1023/A:1005412111706
A. Sterling, L. Abbeduto, Language development in school-age girls with fragile X syndrome. Journal of Intellectual Disability Research. ,vol. 56, pp. 974- 983 ,(2012) , 10.1111/J.1365-2788.2012.01578.X
Marjorie Beeghly, Bedonna Weiss-Perry, Dante Cicchetti, Beyond sensorimotor functioning: early communicative and play development of children with Down syndrome Children with Down Syndrome. pp. 329- 368 ,(1990) , 10.1017/CBO9780511581786.011